Stress behavior deafblind

Research & Practice for Persons with Severe Disabilities 2013, Vol. 38, No. 3, 139–156

copyright 2013 by TASH

Stress, Behavior, and Children and Youth Who Are Deafblind Catherine Nelson University of Utah Robin G. Greenfield University of Idaho Holly A. Hyte and Jason P. Shaffer University of Utah to the forefront senses and processes needed to evaluate and manage perceived threats to well-being (Gunnar & Quevedo, 2007; McEwen & Seeman, 1999; Sapolsky, Romero, & Muck, 2000). Briefly, in response to a perceived stressor, two interrelated systems are activated: the sympathetic adrenomedullary (SAM) system and the hypothalamic-pituitary-adrenocortical (HPA axis) system.

Both are involved in the release of a variety of hormones under the direction of the hypothalamus. The SAM system is the immediate rapid-fire system that, among others hormones, releases epinephrine (adrenaline) and is involved in the fight or flight response. The HPA axis produces a cascade of longer lasting hormones that ends in the production of glucocorticoids (Gunnar & Quevedo, 2007). The effects of glucocorticoids take many minutes or hours to produce and affect physiology and behavior for long periods of time (Sapolsky et al., 2000). Cortisol, a hormone belonging to the glucocorticoid class, distributes glucose to organs that are critical to action in the face of a threat and away from systems not immediately needed.

Therefore, glucose is directed toward the heart and brain and away from the digestive and reproductive systems.

After the perceived threat is deemed to be under control or no longer present, the HPA axis cascade is reversed in a feedback inhibition that results in the cessation of the release of additional cortisol as the organism returns to equilibrium and the crisis passes (Sapolsky et al., 2000).

Cortisol levels vary both on a circadian cycle and reactively in response to an acute stressor (Miller, Chen, & Zhou, 2007; Nicolson, 2007). In a normal circadian (diurnal) cortisol cycle, highest cortisol levels occur upon wakening with a decrease seen over the day. Deviance from this rhythm either in the direction of a hyperactive or hypoactive (flattened) diurnal curve can be seen in individuals experiencing chronic stress (Nicolson, 2007). In the reactive cortisol response to an acute stressor, the cortisol reaches its peak 20 to 30 minutes after the onset of the stressor with a gradual return to baseline over the course of 40 to 60 minutes (Nicolson, 2007; Ramsay & Lewis, 2003).

Perceived stressors may be acute, sequential, episodic, sustained, or anticipated, and each variation will affect the expression and duration of the stress response (Sapolsky

Children and youth who are deafblind with multiple disabilities have several identified risk factors for experiencing toxic levels of stress, and such stress is known to impair physical, mental, and emotional health. This singlecase multiple baseline study examined the frequency and duration of behaviors thought to indicate stress, the duration of active participation in class activities, and the effect of the stress hormone salivary cortisol to determine the extent to which participating students with deafblindness were experiencing stress and how it affected their behavior and participation in school activities. Examined were three groups of intervention strategies designed to decrease stress and its behavioral manifestations and, at the same time, increase active participation. The selected intervention strategies did significantly lower behaviors that had been thought to indicate stress and concomitantly increased active participation. However, sampled salivary cortisol demonstrated only one instance of actual toxic stress. This finding is discussed from the perspective of delineated protective factors against toxic stress.

DESCRIPTORS: stress, deafblind, multiple disabilities, salivary cortisol, attachment Stress is a word common to the modern English vernacular that has come to have numerous connotations and definitions. Viewed from a psychological perspective, stress occurs when an individual perceives challenges as overwhelming when compared to resources and coping abilities (Greenberg, Carr, & Summers, 2002; Gunnar & Quevedo, 2007; Janssen, Schuengel, & Stolk, 2002).

From a biological view, stress is an organism’s digression from a state of homeostasis or equilibrium and its consequent activation of neurobiological systems (stress response) that enables it to return to the point of homeostasis (McEwen & Seeman, 1999).

Stress is a part of everyday life, and the neurobiological stress response is necessary for survival because it brings Address all correspondence and reprint requests to Dr. Catherine Nelson, Department of Special Education, University of Utah, 1705 E. Campus Center Drive, Room 221, Salt Lake City, UT 84112. E-mail: cathy.nelson@utah.edu 139

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et al., 2000). If the stress response is too intense, activated too frequently, or for too long a time, the response becomes toxic and is detrimental to both physical and mental health and the developing brain is particularly susceptible to such toxic stress (National Scientific Council on the Developing Child, 2005). Long-term stress leads to long-term elevation of cortisol levels that can alter the structure of the regions of the brain that are essential for both learning and memory (McEwen & Sapolsky, 1995).

Toxic stress increases the risk of heart disease, diabetes, hypertension, drug abuse, alcoholism, and anxiety disorders (Charmandari, Tsigos, & Chrousos, 2005; Lee, Ogle, & Sapolsky, 2002). In a large national report on mental health problems in early childhood, the National Scientific Council on the Developing Child (2008) stated that toxic stress can impair emotional well-being, early learning, exploration and curiosity, school readiness, and school achievement of the developing child (p. 2). Protective factors against toxic stress that have been delineated in research include (a) the presence of sensitive and responsive caregivers (Gunnar & Quevedo, 2007; Albers, Riksken-Walraven, Sweep, & de Weerth, 2007; National Scientific Council on the Developing Child, 2005, 2008), (b) secure attachment relationships (Gunnar & Donzella, 2002; Spangler & Grossman, 1997), (c) highquality early care and education (National Scientific Council on the Developing Child, 2005; Sims, Guilfoyle, & Parry, 2006; Watamura, Donzella, Alwin, & Gunnar, 2003), (d) peer acceptance (Adam, 2006), (e) responsive environments (Francis, Diorio, Plotsky, & Meaney, 2002), and (f) feelings of competence (Bhagat, 1988; Butcher, Wind, & Bourma, 2008).

Toxic or pathological stress occurs when (a) there is a perceived lack of control, (b) resources are judged to be either not available or ineffective, (c) there is a predominance of negative emotion, and, finally, (d) there is an absence of supportive relationships (National Scientific Council on the Developing Child, 2008). In a meta-analysis of experimental studies, Dickerson and Kemeny (2004) found that social-evaluative threat during task performance and low control over the situation were the two strongest predictors of acute cortisol responses in humans. However, individuals vary greatly in both their perception of stressful events and their vulnerability to them (Gunnar & Quevedo, 2007; Sapolsky et al., 2000; Shonkoff & Phillips, 2000), and although individual temperamental characteristics appear to play a large role in vulnerability to the effects of stress, temperament must be considered in the context of social relationships (Blair et al., 2008; Gunnar & Quevedo, 2007; Shonkoff & Phillips, 2000). Thus, research suggests that temperamentally inhibited children who have had a lack of supportive care are at increased risk for greater stress reactivity (Gunnar & Quevedo, 2007).

For a variety of reasons, children and youth who are deafblind with additional disabilities have been considered to be particularly vulnerable to toxic stress (Chess,

Korn, & Fernandez, 1971; Janssen, Riksen-Walraven, & van Dijk, 2003; McInnis & Treffry, 1982). Communicative behaviors of children who are deafblind are often subtle and difficult to interpret, and as a result, such behaviors are often missed or misinterpreted. Consequently, the children may intensify their signals and cues to express their frustration and mounting stress through self-abusive, challenging, or aggressive behaviors (Hartshorne & Cypher, 2004; Janssen, Riksen-Walraven, & van Dijk, 2002). Children who are deafblind have a limited ability to perceive information through their distance senses of vision and hearing and thus may be unable to anticipate upcoming changes in their environment (Nelson, van Dijk, McDonnell, & Thomson, 2002; van Dijk, Nelson, Postma, & van Dijk, 2010). Such perceived sudden changes in the environment, in the absence of resources to adjust to them, are likely to evoke stress responses (Janssen, Schuengel, et al., 2002). In addition, research has suggested that children who are deafblind are at increased risk for impaired attachment relationships due to a number of factors including frequent hospitalizations, lack of face-to-face eye contact, lack of joint attention, slow, difficult to read responses to caregiver initiations, and difficulties in regulation of arousal patterns (Janssen et al., 2003; Nelson et al., 2002; Nelson, van Dijk, Oster, & McDonnell, 2009; Rowland, 1984). Moreover, chronic stress in children who are deafblind has been thought to manifest itself in physical illness, self-abusive behaviors, and severe challenging behaviors (Janssen, Schuengel, et al., 2002). Professionals in educational settings who are not sensitive to subtle communication cues, reactive to the needs or the children, or who lack skills that promote quality relationships may contribute to the stress experienced by children who are deafblind (Janssen, Schuengel, et al., 2002). There are mechanisms in place nation-wide to train teachers on how to work with children who are deafblind, and there is a large body of research that has documented the effectiveness of various interventions on reducing challenging behaviors experienced by children and youth with multiple disabilities, including deafblindness while increasing participation in activities. Such evidence-based strategies include the following intervention groups: (a) provision of meaningful, reinforcing, and interactive activities (e.g., Brown, Evans, Weed, & Owen, 1987; Downing & Eichinger, 1990; Goetz, 1997; Janssen et al., 2002; Siegel-Causey & Bashinski, 1997; (b) communicative strategies designed to increase anticipation and make environments more predictable (e.g., MacFarland, 1995; Rowland & Schweigert, 2000; van Dijk, Klomberg, & Nelson, 1997; van Dijk, 1996); and, finally, (c) supportive and calming strategies to assist with modulation of biobehavioral state and child regulation (e.g., Guess, Roberts, & Rues, 2002; Nelson et al., 2002; Sterkenburg, Schuengel, & Janssen, 2008). The research base is not clear, however, on the role such interventions might play in reducing stress.

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Cortisol, the end product of the HPA axis, has received much attention from researchers in the area of stress. Salivary cortisol sampling, in particular, has been shown to be an effective and reliable measure of stress and the efficacy of interventions for stress reduction (McCarthy et al., 2009; Nicolson, 2007). It is notably attractive because collection of salivary cortisol does not require a healthcare professional, and samples can be collected in a variety of settings including the educational environment. However, collection of salivary cortisol is but one of many ways to investigate patterns of stress reaction and regulation (Willoughby, Vandergrift, Blair, & Granger, 2007; Ramsay & Lewis, 2003). Because cortisol production and behavioral manifestations progress on different timelines, the collection and tracking of both cortisol and behavioral data yield a more complete representation of stress reactions in individuals than does either in isolation (Ramsay & Lewis, 2003).

The current single-case multiple baseline study utilized an individualized three-part intervention package, designed to reduce stress experienced by children and youth who are deafblind. Both reactive salivary cortisol and behavioral data were collected as the following research questions were examined: (a) Do children and youth who are deafblind and identified as exhibiting stress behaviors actually experience an abnormal level of reactive cortisol in school settings? (b) Do individually determined stress regulation strategies reduce levels of reactive cortisol in individuals who are deafblind? (c) Do individually determined stress regulation strategies reduce behaviors that are purported to indicate stress in individuals who are deafblind? (d) Do individually determined stress regulation strategies increase active participation in school activities? (e) What is the least intrusive level of intervention necessary to reduce stress-like behaviors and increase active participation?

Method Participants and Settings The three participants were children and youth identified as having both vision and hearing impairments and meeting the criteria for inclusion in the National Child Count of Children and Youth Who Are Deaf-Blind (National Consortium on Deaf-Blindness, 2010). All of the participants had multiple disabilities in addition to their vision and hearing impairment and had been identified by their teachers and/or deafblind specialists as experiencing abnormal levels of stress as evidenced by their behaviors. Parents, teachers, and paraprofessionals further delineated and described the behaviors thought to signify stress. The research study was conducted in the children’s school classrooms that were located in urban areas across two western states of the United States, and each of the participants had a one-on-one assistant throughout the school day.

Participant 1, BDavid,[ was a 13-year-old boy who attended general education classes in a middle school.

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He had a profound hearing loss and had recently received a cochlear implant that was worn consistently but was infrequently turned on. He had high myopia or near-sightedness, and his eyes did not work together.

He was able to see and respond to sign language from a 5-foot distance. Additional disabilities included hydrocephalus, an absent corpus callosum, and cerebral palsy on one side of his body; however, he was independently mobile. Additionally, he was identified as having developmental disabilities with academic skills on a first grade level. David was conversant in Signed English and had an interpreter/one-on-one assistant in his inclusive classes. He was able to sign in complete sentences.

Behaviors identified as signifying stress included refusal to participate, closing his eyes, crying, yelling, Bfreezing[ or becoming still, and several diversionary techniques. Behaviors identified as signifying regulation were smiling, laughing, signing Bready[ or Bbetter,[ and actively participating in the ongoing activity. Activities identified as particularly stressful were (a) Physical Education (PE), (b) transition from computer time, and (c) Science.

Participant 2, BMichael,[ was a 4-year-old boy who attended a self-contained special education preschool class. He had a cataract on his right eye and no lens on the left. He was also identified as having cortical visual impairment or CVI. He had been diagnosed as having a functional hearing loss with inconsistent auditory responses. In addition, he had cerebral palsy and was quadriplegic. Michael communicated on a nonsymbolic level, primarily through vocalizations and facial expressions. He was on seven medications for seizure control, reflux, and sleeping. However, none of his medications is known to influence salivary cortisol levels. Behaviors identified as signifying stress were low, continuous vocalizations, teeth grinding, high-pitched vocalizations, crying, continuous grimacing, and pulling away. Behaviors denoting regulation were smiling, active participation, active looking in the direction of an activity or person, and Bhappy[ vocalizations. Identified stressful activities were (a) hand-over-hand activities, (b) having his neck brace put on, and (c) being placed in a wooden box with sensory objects that was termed the Blittle room.[ Participant 3, BAlik,[ was a 6-year-old boy who attended a self-contained class for children with visual impairments and multiple disabilities. He was totally blind and had a severe to profound hearing loss. He had bilateral hearing aids that were seldom worn. In addition, Alik had cerebral palsy and walked with the aid of a walker. He was also identified as having developmental disabilities and communicated on a nonsymbolic level through vocalizations, facial expressions, and body movements. His behaviors thought to indicate stress were resisting or pulling away, yelling crying, sleeping, self-abusive behaviors, and aggressive behavior toward others such as hitting, biting, and pinching.

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Behaviors signifying regulation were becoming still, smiling, laughing, actively participating in the activity, flapping hands, and Baffectionate[ behaviors such as hugging. Activities identified as being stressful were (a) table time activities that included coloring and cutting paper and (b) transition from sensory activities. A third activity termed Bsmall group[ that consisted of a small group of children in the class doing the same activity at the same time was terminated before intervention could be implemented.

Design A multiple baseline across activities design using sequential withdrawal (Kadzin, 1982) was used to evaluate the effects of the three-part intervention package on the dependent variables. The sequential withdrawal design allowed for the determination of the least amount of intervention necessary to reduce behaviors indicating stress and increase active participation, thereby increasing possibilities for child self-regulation. The study was replicated across the three participating children. Independent variables were the following three parts of the evidence-based intervention: (a) environmental strategies directed toward making the activity meaningful, reinforcing, and interactive (e.g., changing the activity to be salient and enjoyable, providing opportunities for choice-making, following the child’s lead, and involving the child in all phases of an activity); (b) adding communicative, anticipatory strategies to ones inherent in the activities (e.g., touch cues, object cues, verbal cues, schedule, or calendar systems); and (c) providing calming strategies (e.g., bringing child to midline orientation, providing vibratory toys, stroking child in a calming manner, and verbally reflecting the child’s emotions). Each child experienced a baseline condition, an Intervention 1 condition with all parts of the intervention package implemented, an Intervention 2 condition with environmental and anticipatory strategies only, and an Intervention 3 condition with only environmental strategies in place. Each child, with the exception of Alik, experienced all conditions across three school activities. Alik experienced baseline in three activities but only had intervention in two. Activities were of different lengths, and to allow for accurate cortisol collection and limit possibilities of intervening stressing events, observations began at transition to the activity and ended when child had been in calm, regulated state indicated by 90% behaviors signifying a regulated state for three consecutive minutes. If such regulation did not occur, the observation continued until transition to the next activity or the student was removed from the activity.

Measurement Dependent measures were (a) percentage of seconds a child engaged in duration-based behaviors (e.g., crying) that were thought to indicate stress, (b) rate of discrete behaviors (e.g., hitting self) thought to indicate

stress per minute, (c) percentage of seconds a child actively engaged in activity, and (d) salivary cortisol levels.

Behavioral data A hand-held tablet computer-based data collection system (Nokia N810) using a custom software application (Nelson & Nelson, 2009) was used to collect all data except salivary cortisol levels. Each data collection tablet device contained customized lists of buttons with each child’s individually determined stress and regulation behaviors. The devices stamped the time in which each answer was entered allowing for point-by-point reliability between data collectors. When duration-based and active engagement behaviors were selected, a timer automatically started. The devices also timed the interval beginning at transition to the activity and ending when the participant exhibited 90% behaviors indicating a calm, regulated state for three consecutive 1-minute intervals. After each session, data were uploaded to a database, and reports were generated using Microsoft SQL Server with Reporting Services.

Salivary cortisol Reactive salivary cortisol was collected for each child on one of three activities thought to be the most stressful. The activity selected for cortisol collection for David was Activity A: PE; for Michael, Activity B: hand-over-hand activities; and for Alik, Activity C: table activities. Collection of saliva was accomplished through the use of a rolled cotton Salivette swab placed in the child’s mouth for 2 minutes or until the swab was saturated. Cortisol was collected at the same time each day and at least 60 minutes after a child had eaten or had anything to drink other than water. The swabs were frozen immediately after collection and remained on ice until delivery to ARUP Laboratories at the University of Utah in Salt Lake City, Utah. Samples were collected by the co-principal investigators in the two states. To capture the reactive cortisol curve, salivary cortisol was collected immediately preceding the activity thought to be stressful, 20 minutes after onset of the activity, and then again at 40 minutes post onset.

Experimental Design and Conditions Baseline Before implementation of the new interventions, researchers used the tablet computers to record observations on the three selected activities in each child’s classroom for several days to establish a baseline of the child’s behaviors. Baseline probe data were minimally taken for three consecutive school days before beginning intervention on each of the dependent measures. Intervention began when the baselines of stress-like behaviors were stable within each activity. Cortisol samples were taken during one activity, three times per collection session as outlined above during a minimum of two sessions: one collection session at the beginning of baseline and one the day before intervention one began.

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Intervention Observations of all the participants were conducted three times weekly in all intervention conditions because of school schedules and excluded holidays or other special events. Michael only attended preschool three times per week, and the transition activities for both David and Michael only occurred 3 days per week. All behavioral data collected in baseline were collected in all three intervention conditions and in identified activities as delineated in the Procedure section. Reactive salivary cortisol was collected as outlined above during one selected activity in each of the three intervention phases. These collections occurred on three consecutive data collection days immediately preceding the onset of the next phase or, in the case of Phase 3, before intervention in the phase terminated.

Interobserver Agreement and Fidelity The first author of the study conducted training on data collection methods with research personnel from the two states. Using a video-taped example of three activities of a child who was not a part of the study, all data collectors practiced data collection with the tablet devices until they reached 90% or better reliability with the first author of the study. Primary data collection was done by the two first authors of the study. Secondary data collectors were research assistants identified by each university as having experience and expertise with children who are deafblind.

Primary and secondary data collectors collected data simultaneously during 25% of sessions across all conditions (baseline and Interventions 1, 2, and 3). The point-by-point formula used was agreement divided by agreement plus disagreement multiplied by 100 (Kadzin, 1982). A tolerance of plus or minus 5 seconds was set as an acceptable level of agreement for each event and all of the behavioral dependent measures were included in the reliability measure. Mean agreement for David was 98% (mean range of agreement in baseline, 89Y100% and range across the three inventions, 90Y100%). Mean agreement for Michael was 96% (mean range in baseline, 82Y100% and mean range across interventions, 85Y100%). Mean agreement for Alik was 93% (mean range in baseline, 80 to 100%, mean range across interventions, 80Y100%). Teachers and one-on-one assistants received individual training and guided intervention shortly before new intervention began, and then condition requirements were reviewed before each condition change. Classroom teachers were involved in the design of environmental modifications and strategies in both baseline and intervention conditions, but day-to-day interventions were implemented by the one-on-one assistants. A research assistant observed each intervention during 20% of sessions across all intervention phases to monitor fidelity to outlined intervention procedures. This was accomplished through the use of a checklist individually prepared for each child’s inter-

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ventions. Fidelity data were calculated by dividing the number of intervention behaviors exhibited by the number of preplanned behaviors and multiplying by 100.

Mean treatment fidelity for David was 100% across phases and activities, 95% for Michael (range, 88Y 100%), and 98% for Alik (range, 96Y100%). Lower fidelity was seen in a lack of wait time in the first phase of intervention, and interventionists were prompted to increase the wait time until 100% was achieved.

Procedure Intervention 1 When participant achieved a stable baseline in the first activity (Activity A), Intervention 1 began. This condition included all three components of the intervention package: (a) making the activity meaningful, (b) anticipatory strategies, and (c) calming strategies. When visual analysis of the data revealed a trend decrease in stresslike behaviors, Phase 1 was implemented in Activity B. Intervention Phase 1 began in Activity C when child reached target in Activity B. Intervention 2 When participant met target for Intervention 1, Intervention 2 began. Intervention 2 was comprised of the first two components of the intervention package: (a) making the activity meaningful and (b) anticipatory strategies. Calming strategies were not used in this phase unless the child failed to reach a calm state within 10 minutes. Intervention 2 was implemented in all three activities as described above in Intervention 1.

Intervention 3 When participant met target for Intervention 3, Intervention 3 was implemented. Intervention 3 was comprised of meaningful activity only, and procedures were as outlined under Intervention 1.

Individual Interventions Individuals who are deafblind with additional disabilities are a diverse group and so too were the participants in this study. School settings and activities also varied and included both self-contained special education and inclusive classes across preschool, elementary, and secondary settings. Although unique to each individual and activity, the strategies used were exemplars of each of the three parts of the intervention package. The package was based on the evidence-based research deemed effective in reducing challenging behaviors and increasing participation in activities that are delineated above in the literature review. Baseline activities, selected interventions, and definition of active participation for the particular activity are described for each participant below.

David Baseline (Activity A: PE) PE took place in a junior high general education PE class. During baseline, David participated by rolling a ball back and forth with his interpreter/one-on-one

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assistant on the sidelines of the gym. He was dressed in his street clothes and often looked at the other students and vocalized loudly.

Intervention To make the activity more meaningful, David was provided with a gym suit like that of other students in the class, which he put on in the locker room with his peers.

He participated in all PE activities with his peers that generally consisted of warm-up exercises followed by sports such as flag football and dodge ball. He was told to follow what the other students were doing, and peers, without prompting, assisted him to be successful as needed. Because of health issues, David took 5-minute breaks when he became tired. Anticipatory strategies were an activity picture schedule for each of the activities in PE and a general picture schedule for afternoon classes. His interpreter reviewed each step on the schedule with him before class began and redirected him to the schedule if he got off task. Calming strategies consisted of his interpreter, leading him back to the activity and staying within 5 feet of the activity until he was in a calm state.

Active participation Active participation was defined as participating in the activity as requested (i.e., rolling ball with interpreter in baseline and participating in same activities as peers in intervention with the exception of 5-minute breaks).

Baseline (Activity B: Computer transition) Before Science class, David had a 15-minute break in which he was allowed to play games on the computer.

When it was time to go to class, he was informed that time was up. He would frequently become very agitated and refuse to turn off the computer and get ready to go. Therefore, he was frequently very late for Science and would still be exhibiting stress-like behaviors when he got there.

Intervention To make the activity more meaningful, computer activities and games related to content being studied in Science were selected, and David’s choices of computer activities were limited to the identified science-related activities. At the onset of the activity, he was given a 10-minute time limit. His agitated behaviors decreased during intervention but were still unacceptably high, and therefore, tangible reinforcement (one piece of candy) was added at Day 4 of the Intervention 1 condition, contingent on shutting down the computer, putting on his backpack, and starting to walk to Science. Anticipatory strategies consisted of an activity picture/word schedule of his day and a similar schedule that included the steps of the computer activity, which his interpreter reviewed with him. He also had a 3-minute hourglass that was turned over to let him know he had 3 minutes left in the activity. If he became agitated during the transition,

the computer was turned off and the calming strategy of being allowed to remain sitting in his seat with no further direction or attention from his interpreter until he signed Bready[ or got his backpack on was implemented.

Active participation Active participation was defined as shutting down the computer, putting backpack on, and walking to class.

Baseline (Activity C: Science) During baseline, David was seated at the back of the general education science class with his interpreter and looked at books unrelated to science. He frequently made loud, disruptive vocalizations. His bus came before the end of the session, and hands-on laboratory activities generally occurred after he had left.

Intervention To make the activity meaningful, David was seated close to the front of the room so that he could see the teacher and be with his peers. His interpreter was seated facing him. At the beginning of class, the interpreter reviewed with him his classroom rules of sitting, watching the teacher, being quiet or participating, and finally, leaving his cochlear implant turned on. She interpreted information the teacher gave at a simplified, general level. David was given check marks, which led to tangible reinforcement for following the rules. The classroom teacher ensured that labs occurred early so that he did not miss them. Anticipatory strategies included an activity picture/word schedule that included pictures of his classroom rules. Check marks were made on the schedule. If he became tense or upset, the calming strategy implemented was having the interpreter place her hand atop his.

Active participation Active participation was defined as leaving cochlear implant turned on, looking at teacher and interpreter, looking at class materials, and/or appropriately engaging with materials and classmates in class activities.

Michael Baseline (Activity A: Neck brace) Michael’s assistant put a large foam rigid neck brace on Michael every morning. His wheelchair was in an upright, locked position, and without warning of what was to come, the assistant would move his head forward and put the brace on. In response, Michael would grimace and cry, and the assistant would either not respond or tell him he was BOK.[ Intervention In an attempt to make the activity more meaningful as well as comfortable, Michael’s wheelchair was slowly put into a reclined position, and the brace was moved into his left visual field so that he could see it. He was guided through the use of hand-under-hand assistance

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to touch the brace as it was put on. Anticipatory strategies of verbal cues and a tactile cue of a tap on the back of his head were provided. Comforting strategies were rocking his wheelchair back and forth, stroking his head, and having his assistant use a soothing voice.

Active participation Active participation was defined as fixating visual gaze on neck brace when it was slowly moved in his left peripheral field, keeping left hand on the brace after being guided to touch it and remaining quiet or exhibiting regulation behaviors when brace was put on.

Baseline (Activity B: Hand-over-hand activities) In this activity, Michael was expected to do the fine motor activities such as coloring and cutting that the other children were doing. Because he had no functional use of his hands, his assistant used complete hand-overhand assistance. He was given no prior information to allow him to know what was going to happen and because of his visual and motor limitations could not see what he was doing. In response, Michael would pull back and emit low continuous vocalizations.

Intervention To make the activity meaningful, Michael was given a choice of two tactile activities (e.g., strumming a toy guitar), which he chose through eye gaze or keeping his hand on the object. His wheelchair position was adjusted for visual and tactile interaction with the objects. He was guided to explore materials through hand-underhand exploration. Anticipatory cues included the visual cue of the object and the verbal cue of Blet’sI.[ If he seemed upset, he was comforted by his wheelchair being rocked back and forth, verbal soothing, and deep pressure massage.

Active participation Active participation was defined as allowing hands to be moved without pulling back, keeping hand on an item or person, moving hand toward an object, exploring objects with his fingers/hand, and initiation of movement.

Baseline (Activity C: Little room) This activity was an interpretation of a strategy known as the Blittle room,[ which has been used with children with visual impairments to encourage development of spatial relationships and independent exploration of motivating materials in a confined space in which materials are close at hand (Nielsen, 1992). In this interpretation, the Blittle room[ was a large box approximately 3 inches by 2 inches that was open at the foot and had a clear top. Unfamiliar objects were placed in the box, and the activity occurred in a dark closet. After placing Michael in the box, the assistant stood outside the closet. Generally, Michael’s reaction was to cry and engage in highpitched vocalizations until he was removed.

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Intervention To make the activity more meaningful, an open Blittle room[ made with a PVC pipe framework with items of interest hanging from the pipes was constructed.

Michael was slowly placed under the structure, on a mat with a pillow under his head, and lights were dimmed.

Anticipatory strategies were visual and verbal cues given immediately before the activity, presentation of red bell that was also inside the Broom,[ and assistance to feel the mat. Calming strategies were verbal soothing and having his legs and feet massaged by his assistant who stayed at his side.

Active participation Active participation was defined as fixating gaze on red bell presented in his left visual peripheral field, not resisting or pulling back from hand-under-hand guidance of his left hand when it was moved to touch the bell and vinyl mat, visually fixating on items that were moving in his visual field, and touching items that were placed near his left hand.

Alik Baseline (Activity A: Table time activities) In this activity, Alik was lifted into an ill-fitting wooden chair and confined with a seatbelt. His hearing aids were not put on, and all information was given verbally. His assistant used hand-over-hand assistance in group table activities that included coloring and cutting with scissors, which he was unable to see because of his blindness.

Alik engaged in most of his behaviors thought to indicate stress immediately upon being put in the chair.

Intervention To make the activity meaningful, it was moved to a comfortable space on the floor so that Alik could move around freely and his hearing aids were put on. Activities were changed to reflect his goals and included listening and movement activities with musical instruments and turn-taking musical games. He was given choices of activities through bins containing activity materials. Anticipatory cues included verbal cues and feeling the activity bin before going to the activity. Calming strategies were hugs, arm stroking, and the provision of comfort items such as a vibrating pillow.

Active participation Active participation was defined as reaching for and manipulating materials and performing steps of the activity.

Baseline (Activity B: Transition from favored activity) Alik would frequently become very agitated when he was transitioned from a sensory activity such as water play. Therefore, baseline was conducted during a consistent water activity in which a small tub filled with water was placed atop a mat on the floor, and Alik splashed and played with toys in it. This was followed by taking him to

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the sink to wash his hands. No warning of the upcoming activity change was provided, and there was no predictable sequence of activity. Alik responded by crying, hitting his head, and trying to bite and pinch his assistant.

Intervention To make the activity more meaningful, a consistent activity sequence was implemented, and Alik was assisted to be involved in all activity phases, from preparing the materials in the water activity through putting them away.

As he moved to the sink, his assistant sang a consistent transition song into his hearing aid. He was then given a choice of materials to use in the next activity. Anticipatory strategies were bins with materials from each part of the activity arranged in sequence (e.g., paper towel to signify hand washing in final bin) and provision of a 1-minute verbal warning along with assistance to again feel the next activity bin. Calming strategies were hugs, arm stroking, and vibrating toys.

Active participation Active participation was defined as reaching for activity bins and touching materials contained in each, actively assisting in clean-up of materials, walking with assistance to sink to wash hands, and walking with assistance to next activity.

Activity C (small group) In this activity, which was terminated before the end of baseline, Alik sat in his wooden chair and was asked to do curricular tasks such as counting blocks, measuring various items, and making holiday crafts. All children in the group were asked to do the same task at the same time.

Data Analysis The dependent variables of duration-based stress-like behaviors, discrete stress-like behaviors, and time spent actively engaged in activities were plotted on multiple baseline graphs for each child (see Figures 1Y6). Data

Figure 1. David. Left ordinate represents the percentage of seconds that the child engaged in duration-based stress-like behavior.

Right ordinate represents the rate of discrete stress-like behaviors per minute.

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Figure 2. Michael. Left ordinate represents the percentage of seconds that the child engaged in duration-based stress-like behavior.

Right ordinate represents the rate of discrete stress-like behaviors per minute.

were examined after each session by researchers using visual analysis for changes in level, latency of change between phases, and changes in trend within phases.

Salivary cortisol was analyzed at ARUP Laboratories at the University of Utah using Enzyme Immunoassay, and the coefficient of variation on the assay was G8%.

Reference intervals for age groups, gender, and time of day were established by ARUP laboratories on nonCushing disease populations. Salivary cortisol levels of the participants were then compared to the reference intervals and plotted on graphs (see Figure 7).

Results Data presented in this section report the impact of the different intervention conditions on dependent variables for each child across activities. Summarized daily data of discrete and duration-based stress-like behaviors

are provided in Figures 1, 2, and 3. Active participation data are provided in Figures 4, 5, and 6. Provided in Figure 7 is the comparison of the salivary cortisol data for David, Michael, and Alik to established reference norms to answer the research question of whether the children who exhibited stress-like behaviors actually were experiencing toxic stress.

David As seen in Figure 1, David demonstrated a significant downward trend of duration-based stress-like behaviors from baseline through all intervention conditions. No effect could be shown for the discrete stress-like behaviors because of the low baseline levels. In Activity C (Science), the last day of data collection was the last day of school, and although avoidance behaviors went up, they were not significantly different from those of his classmates.

The removal of calming strategies (Intervention 1) in PE

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was initially difficult when the interpreter moved further away from him but otherwise, across activities, the removal had little impact. In all of the activities, David concurrently signed the pictured steps in his schedule systems that comprised his anticipatory strategies and continued to sign them without prompting in intervention Phase 3 when the pictures were no longer available.

It might therefore be assumed that he had internalized the schedule system, and its removal did not cause an increase in the behaviors. Stress-like behaviors persisted in Intervention Phase 1 of Activity B (transition from computer), and on the fourth day of intervention, the decision was made to implement tangible reinforcement

to make the transition more reinforcing than the computer activity. There was an initial burst of stresslike behaviors the following day, but then a decrease thereafter. However, duration-based stress-like behavior did not change in Activity B until about the same time it changed in Activity C. Active participation is graphically displayed in Figure 4; in general, active participation went up between all baseline and intervention conditions.

Mean participation in baseline was 52% (range, 38Y 64%), in Intervention 1, 83% (range, 36Y99%), in Intervention 2, 88% (range 47Y100%), and in Intervention 3, 90% (range 65Y100%). The lowest participation percentage in Intervention 3 was the last day of school, and

Figure 3. Alik. Left ordinate represents the seconds that the child engaged in duration-based stress-like behavior. Right ordinate represents the rate of discrete stress-like behaviors per minute.

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Figure 4. David. Percentage of seconds the child actively engaged in activity.

again, his participation was not significantly different from that of his peers.

Salivary cortisol For the most part, as seen in Figure 7, reactive salivary cortisol levels in PE were within normal reference intervals for age and time of collection during baseline and all three intervention conditions. There were two instances when the levels were nominally above normal, and both occurred on days in which David vigorously participated in flag football. Sustained, intense physical activity is known to raise cortisol levels (Jacks, Sowash, Anning, McGloughlin, & Andres, 2002), and given David’s baseline behavior and physical disabilities, it was rather unexpected that he would participate to an extent that would cause even such a slight increase. Toxic stress was not, however, demonstrated in any of the samples.

Michael As seen in Figure 2, the percentage of each observation that Michael engaged in duration-based stress-like behaviors decreased substantially from baseline though the Intervention 1 condition. The frequency that discrete behaviors occurred was more variable, but also trended down. Activity A was having his neck brace put on, and this was a difficult activity for Michael. Stress-like behaviors went up when calming strategies were removed.

Michael had many health issues, and although it meant beginning intervention in Activity B (hand-over-hand manipulation activities) when stress behaviors appeared to be trending downward, it was felt that baseline levels remained unacceptably high. When intervention began, stress-like behaviors showed an immediate, marked drop, which was sustained throughout the phases. In Activity C or the Blittle room[ activity, stress-like behaviors

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again went dramatically down between baseline and Intervention 1 with a slight burst occurring the first day of Intervention 3 when anticipatory strategies were removed. The percentages went back down when it is presumed he adjusted to the new routine. Therefore, a modest effect was demonstrated for discrete stress-like behavior, and experimental effect was documented for duration-based stress-like behavior. Across activities, active participation trended substantially upward as seen in Figure 5. Mean participation in baseline across activities was 8% (range, 0Y41%), in Intervention 1, 55% (range, 0Y94%), in Invention 2, 66% (range, 1Y93%), and in Intervention 3, 76% (range, 42Y98%). However, intervention decisions were made based on stress-like behaviors rather than active participation, and there-

fore, intervention was implemented for Activity C even though active participation was on an upswing. Thus, it is difficult to say with certainty that active participation would not have gone up without the intervention.

There is, however, a considerable and consistent change in the magnitude of active participation, and therefore, modest experimental effect can be claimed.

Salivary cortisol Reactive salivary cortisol levels were consistently within normal reference intervals for age and time and day.

Although Michael experienced seizures on several days, salivary cortisol levels did not concomitantly rise. Therefore, toxic stress was not observed. Because of his generally flat cortisol levels, it would be of interest to sample

Figure 5. Michael. Percentage of seconds the child actively engaged in activity.

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Figure 6. Alik. Percentage of seconds the child actively engaged in activity.

Michael’s diurnal cortisol to determine if an abnormally flattened diurnal curve might be present which could suggest a lessened ability to mount a stress response.

Alik As summarized in Figure 3, Alik’s duration and discrete, frequency-based stress-like behaviors decreased between baseline and all three intervention conditions.

In Activity A (table activities), stress-like behaviors did rise in Intervention 3, although behaviors were still below baseline levels. In Activity B, there was a slight rise in behaviors in both Interventions 2 and 3. In both conditions, the final point was at or close to 0%, suggesting he may have adjusted to the condition. On the third day of Activity A, Intervention 3 (also the

midpoint of Activity B Intervention 2), Alik’s oneon-one assistant left her position. Stress-like behaviors recorded in Activity A on the day she left were low (1%), but Alik did persist in trying to sleep, which was a behavior that had been noted as possibly stress related in initial observations but had ceased when the one-on-one assistant started her position just prior to baseline. Activity B did not occur on the day the assistant left. Salivary cortisol results for this day are detailed below in the Salivary Cortisol section and can be viewed in Figure 7. Alik’s new one-on-one assistant received additional training on the strategies before the next session and a familiar speech therapist assisted with the activities. As summarized in Figure 6, active participation increased from baseline in all three intervention

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Figure 7. Salivary cortisol levels compared to reference interval across conditions. High and low values for reference interval are provided. *Denotes break in the y axis on Day 11 to accommodate large variation in data points.

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conditions. Mean active participation in baseline across the two activities was 15% (range, 2Y33%), in Invention 1, 63% (range, 19Y100%), in Intervention 2, 87% (range, 60Y91%), and in Intervention 3, 59% (range, 35Y99%). The highest score in the Intervention 3 condition was seen on the last day of intervention in Activity A. In addition, the occurrence of behaviors indicating stress and active participation varied more after Alik’s familiar one-on-one assistant left. Unfortunately, because of the termination of Activity C before intervention could begin, experimental control is limited. However, it should be noted that the baseline behaviors in Activity C did not vary with the onset of intervention in Activities A and B. The claim can be made that Alik’s data are consistent with those of David and Michael, and thus, replication of the basic pattern was seen.

Salivary cortisol Alik’s reactive salivary cortisol levels as displayed in Figure 7 were variable, although they were within the normal reference range for age and time of collection with the exception of two collection sessions. On the first such collection session, cortisol levels were within the reference range on the first two samples, but then went slightly above the range on the third collection. Alik’s stress-like behaviors were generally very low on that day (1%), but the brief period of stress behaviors that did occur was at the end of the session, suggesting that something may have interfered, which caused some stress.

On the day Alik’s one-on-one assistant left her position, cortisol levels had a marked increase. Levels were 6.1 on first collection, 915 on the second, and 6.03 on the third, which are all levels well above the upper reference level.

In fact, the second collection was higher than what the laboratory could measure. These levels demonstrated a normal reactive curve for the reactive stress response with the second point being the peak of cortisol. Therefore, abnormally high, toxic stress levels were seen on the day the one-on-one assistant left. The only stress-like behaviors observed during this particular session were Alik’s attempts to sleep. Subsequent cortisol samples were within the normal reference interval.

Discussion Physiological Stress and Behavior Although it has been assumed that children and youth who are deafblind are vulnerable to experiencing toxic stress, the results of the salivary cortisol sampling in this study, with one very notable exception, suggest otherwise. Although all three participants were identified by their teachers as seeming to be Bstressed[ and all had high levels of behaviors suggesting agitation, true stress was only seen on one day and in one participant across baseline and intervention conditions. This finding leads to several conclusions. The first is that true physiological stress is an internal construct that may not manifest itself in external behaviors. On the occasion when Alik did

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have extremely high cortisol levels, his behavior qualitatively changed from more acting out behaviors to a more internal strategy of attempting to sleep.

In revisiting the protective factors against toxic stress, it can be argued that, despite their deafblindness, the participants may have been protected against such stress by both the presence of supportive, responsive individuals with whom they had secure attachment relationships and the control they were able to exercise over their situations. The one instance of abnormally high stress occurred on the day Alik found his one-on-one assistant, with whom he had a very secure relationship, no longer available. In the absence of someone he felt could and would respond to his communications, Alik stopped actively communicating through his behaviors and attempted to sleep; a strategy he had often used successfully before this particular relationship was available to him. His salivary cortisol levels did return to normal the next day as did his behavior when his substitute assistant implemented familiar strategies and routines and responded to his positive communication attempts. In addition, across children and activities, the behaviors suggesting stress began at the onset of the activities and the consequence was often either being removed from the activity or allowed to not participate.

Therefore, teachers and researchers felt that one of the major functions of the behaviors might have been escape, and in baseline, such behaviors were frequently successful in achieving this aim. This perceived success may have provided them a feeling of control over their environments. Therefore, the stress-like behaviors may have served a successful communicative function that was actually a protection against toxic stress rather than a manifestation of it.

Impact of the Intervention Strategies on Behavior and Participation Although the stress-like behaviors may have been successful in achieving avoidance, they were interfering in the participants’ ability to actively participate and learn in their educational settings and activities. They were also often perceived as negative by their teachers and assistants. The intervention strategies in this study achieved the goal of substantially decreasing the frequency of behaviors indicating stress or agitation while, at the same time, increasing active participation. In addition, there was not a concomitant increase in physiological stress when active participation in activities increased.

There was a marked decrease in agitated behaviors as well as an increase in active participation when the strategies of making activities meaningful and interactive were implemented. Given meaningful activities, calming strategies were generally not needed except in the case of Michael’s neck brace that likely remained uncomfortable and not particularly meaningful to him.

Anticipatory strategies appeared important at least initially for all participants, but through the provision of

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meaningful, consistent activities, students were actively involved throughout activity phases and, therefore, in some activities, appeared to access more natural cues inherent and highlighted in the activities and, in some cases, learn the consistent activity sequence. Such natural cues are important because added anticipatory cues may be inconsistently provided. Additionally, the body of stress research has demonstrated that timing of presentation of anticipatory strategies is critical. They are only effective if presented sufficiently before the stressor to allow for the formulation of a response but not so much so that the two events are not seen as connected.

Anticipatory strategies are also most effective when the individual has control of events and are not helpful if the stressing event is deemed to be too severe to manage (Sapolsky, 2004). In the current study, added anticipatory strategies remained necessary in difficult activities such as when Michael’s neck brace was put on and when Alik transitioned from a favored activity. They were also important when changes or modifications to routine occurred. Therefore, it is suggested that level of intervention should be looked at in terms of both the individual child and individual activity and situation.

Limitations In addition to ones mentioned in the Results section, there are some limitations to this study that could affect the extent to which the results can be generalized to other children and settings. The first is that, although the study took place in varied settings, activities, and with different age groups, the number of participants was small and without systematic replication, it is difficult to say that these children and youth as well as their educational settings were representative of the population of children and youth who are deafblind and the settings in which they are receiving education. Second, because the students and activities varied widely, interventions also varied. Although they were within intervention classes, such differences introduced a confounding variable to the comparison of individual interventions. Third, Alik was only able to experience intervention in two activities because of a major change in the classroom. As a part of this change, his one-on-one assistant left, and he had a different assistant on the last few days of intervention in the remaining activities.

The final major limitation was difficulty measuring reactive stress within classrooms. Although the cortisol samplings followed protocol for reactive stress collection, it was impossible to control for interfering variables between sampling. For example, between the second and third samples, at which point cortisol levels should have been returning to baseline, a new, stressful event might have occurred. Cortisol samples could also only be taken 60 minutes after anything to eat or drink other than water, which limited the activities in which cortisol could be collected. Finally, there were potential participating children who were reported to have many stress-like be-

haviors but who also had oral aversions or other behaviors that made collection of salivary cortisol impossible.

Thus, the pool of potential participants was very small.

However, despite these limitations, the study does offer several implications for further research and practice.

Implications for Further Research The results of this study, although somewhat surprising, do parallel a recent study done in the Netherlands (Bloeming-Wolbrink et al., 2012) that examined the diurnal salivary curve of adults who were deafblind and had additional disabilities and found an essentially normal curve indicating normal levels of stress. More research is needed to discover if, indeed, stress is independent of the disability. There might also be differences between the stress levels of individuals with acquired deafblindness and those with congenital deafblindness such as the participants in this study who had been deafblind from birth and therefore might not have found the lack of vision and hearing to be stressful.

More research is also needed in the area of manifestations of stress. Teachers (and parents) assumed that challenging and/or agitated behaviors were indicators of stress, but the assumption was not confirmed in this study. Finally, examinations of the efficacy of individual strategies in both the environmental and anticipatory groups would be useful to the field.

Implications for Practice The importance of providing meaningful, predictable activities with opportunities for interaction and initiation was a clear finding of this study, and such activities can result in higher active participation and increased behavioral regulation. Importantly, the results of the cortisol testing and behavioral data suggest that teachers and other professionals in the field should be mindful of the importance of attachment relationships and their impact on students. The results provide impetus for planned relationships across adults and peers in educational settings and careful transitions for times when individuals with whom a child might have an attachment relationship leaves. Finally, the study suggests that the individuals in this study were capable and resilient. All had been through significant adversity in their young lives and had found successful ways to cope. However, as with all students, positive support is needed if they are to achieve their potential in educational settings.

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